fluence of an Intensive Speech Therapy Program on the Speech ... - Thieme

Article published online: 2022-08-10

THIEME

Original Research

In?uence of an Intensive Speech Therapy

Program on the Speech of Individuals with Cleft

Lip and Palate

Laura Katarine F¨¦lix de Andrade1

Jeniffer de C¨¢ssia Rillo Dutka2

3

Maria Daniela Borro Pinto

Maria In¨ºs Pegoraro-Krook2

1 Program in Rehabilitation Sciences, Hospital of Rehabilitation of

Craniofacial Anomalies, University of S?o Paulo, Bauru, SP, Brazil

2 Department of Speech-Language Pathology and Audiology, Bauru

School of Dentistry, University of S?o Paulo, Bauru - SP, Brazil

3 Speech Department, Hospital of Rehabilitation of Craniofacial

Anomalies, University of S?o Paulo, Bauru, SP, Brazil

Gabriela Zuin Ferreira2

Address for correspondence Laura Katarine F¨¦lix de Andrade, PhD

Student, Hospital of Rehabilitation of Craniofacial Anomalies,

Universidade de S?o Paulo, Rua Silvio Marchiore, 3-20, Bauru (SP),

Brasil 17012-900 (e-mail: lkf.andrade@).

Int Arch Otorhinolaryngol 2023;27(1):e3¨Ce9.

Abstract

Keywords

? cleft palate

? velopharyngeal

insuf?ciency

? speech

? speech therapy

received

July 10, 2020

accepted after revision

December 21, 2020

Introduction Compensatory articulations are speech disorders due to the attempt of

the individual with cleft palate/velopharyngeal dysfunction to generate intraoral

pressure to produce high-pressure consonants. Speech therapy is the indicated

intervention for their correction, and an intensive speech therapy meets the facilitating

conditions for the correction of glottal stop articulation, which is the most common

compensatory articulation.

Objective To investigate the in?uence of an intensive speech therapy program (ISTP)

to correct glottal stop articulation in the speech of individuals with cleft palate.

Methods Speech recordings of 37 operated cleft palate participants of both genders

(mean age ? 19 years old) were rated by 3 experienced speech/language pathologists.

Their task was to rate the presence and absence of glottal stops in the 6 Brazilian

Portuguese occlusive consonants (p, b, t, d, k, g) distributed within several places in 6

sentences.

Results Out of the 325 pretherapy target consonants rated with glottal stop, 197

(61%) remained with this error, and 128 (39%) no longer presented it. The comparison

of the pre- and posttherapy results showed: a) a statical signi?cance for the p1, p2, p3,

p4, t1, k1, k2 and d6 consonants (McNemar test; p < 0.05); b) a statistical signi?cance

for the p consonant in relation to the k, b, d, g consonants and for the t consonant in

relation to the b, d, and g consonants (chi-squared test; p < 0.05) in the comparison of

the proportion improvement among the 6 occlusive consonants.

Conclusion The ISTP in?uenced the correction of glottal stops in the speech of

individuals with cleft palate.

DOI

10.1055/s-0041-1730300.

ISSN 1809-9777.

? 2023. Funda??o Otorrinolaringologia. All rights reserved.

This is an open access article published by Thieme under the terms of the

Creative Commons Attribution-NonDerivative-NonCommercial-License,

permitting copying and reproduction so long as the original work is given

appropriate credit. Contents may not be used for commercial purposes, or

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licenses/by-nc-nd/4.0/)

Thieme Revinter Publica??es Ltda., Rua do Matoso 170, Rio de

Janeiro, RJ, CEP 20270-135, Brazil

3

4

Influence of an Intensive Speech Therapy Program

Andrade et al.

Introduction

The treatment of velopharyngeal dysfunction (VPD) due to

the failure of primary palatal surgery in individuals with cleft

palate, when there are speech symptoms, may require physical and/or behavioral treatment, depending on the etiology

of the VPD.1,2 Physical procedures, such as surgery (secondary palatoplasty or pharyngoplasty, for example) or prosthesis (pharyngeal bulb), when the VPD was caused by

structural anomalies, that is, when there is a lack of tissue

in the soft palate or too much nasopharyngeal space preventing velopharyngeal closure (velopharyngeal insuf?ciency). Speech therapy is indicated when the cause of VPD is

functional, that is, when there is a learning error in the use of

velopharyngeal structures.1 In VPD related to cleft palate, the

same individual may present both insuf?ciency (anatomical

or structural defect that prevents adequate velopharyngeal

closure) and mislearning (an articulation disorder in which

there is a substitution of a pharyngeal or nasal sound for an

oral sound), and for this reason, the combination of physical

and functional treatment approaches is necessary.3

The presence of VPD after surgical correction of cleft

palate can lead to the development of speech alterations

involving both passive and active errors.4 Passive speech

errors are due to an abnormal velopharyngeal structure,

including hypernasality, nasal air emission, and weak intraoral air pressure.5 Active speech errors involve alteration of

articulation placement in response to an abnormal structure,

and this compensatory behavior occur in the attempt of the

speaker to generate and/or maintain adequate levels of

intraoral pressure to produce high-pressure consonants.5

Although these compensatory speech behaviors may be

considered strategies developed in order to achieve the

special requirements of a speech regulating system in the

presence of VPD; acoustically, these responses tend to undermine rather than improve speech performance.6

The incidence of compensatory articulations (Cas) as

described in the literature varies between 6 and

63%.7¨C10The effect of erroneously learned neuromotor patterns used during atypical placement production may dominate the phonological development of the child, creating a

restricted phonetic repertoire that may persist regardless of

the establishment of the potential for velopharyngeal closure. Therefore, it is common for the CA to be incorporated

into the speech of the child, to the point of compromising

his/her speech intelligibility. Even when the palate is restored with a successful management of velopharyngeal

insuf?ciency, these learned behaviors may persist, always

requiring speech therapy to learn an adequate placement

and manner of articulation.11

Glottal stop (GS) is the most common type of CA found in

cleft palate speech.10,11 When this active speech error

becomes habituated and incorporated into the phonological

system of the individual, it can be particularly resistant to

change even during speech therapy.12 The dif?culty of some

clinicians to identify this error may lead them to use inadequate strategies, such as blowing exercises and other activities unrelated to speech to correct the speech error. Besides

International Archives of Otorhinolaryngology

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No. 1/2023

the selection of an adequate therapeutic approach to correct

CA errors, the frequency of speech therapy might also be an

important aspect related to the success of the intervention.

As described in the literature, the hypothesis of the present

study is that a structured intensive speech therapy program

(ISTP) meets the facilitating conditions for the correction of

CA related to cleft palate and VPD.12¨C14

Objective

The present study investigated the outcome of an ISTP to

correct the use of glottal stop productions in the speech of

individuals with cleft palate.

Material and Methods

The present retrospective study was approved by the Ethics

and Research Committee on Human Subjects of the Hospital

of Rehabilitation of Craniofacial Anomalies (1.397.124),

where the present study took place. Informed verbal consent

was obtained from all participants.

Speakers and Speech Sample

The audio recordings used in the present study were retrieved from the database of the Hospital of Rehabilitation of

Craniofacial Anomalies. The samples of interest were

obtained from 37 operated cleft lip and/or palate patients

presenting with VPD, 16 females (43%) and 21 males (57%),

with ages ranging from 6 to 39 years old (mean:19 years old;

standard deviation [SD]: 10.8 years old). All of them were

Brazilian Portuguese speakers who had participated for the

?rst time in one of the modules of ISTP conducted at the

Hospital of Rehabilitation of Craniofacial Anomalies, between 2013 and 2016. The ISTP module involves 45 therapy

sessions lasting 50 minutes provided within a period of

3 weeks ( 3 daily sessions, from Monday to Saturday),

applied by different speech therapists using a combination

of phonological and phonetic speech therapy approaches.

Out of the 37 patients, 34 (92%) used a temporary pharyngeal bulb prosthesis to establish potential for velopharyngeal closure during speech therapy, while the remaining 3

(8%) achieved velopharyngeal closure without a prosthesis or

pharyngeal ?ap.

The speech recordings were obtained pre- and post-ISTP

in a sound-protected environment with high-quality microphones. The recorded speech samples consisted of 6 sentences, each of them with recurrence of the 6 Brazilian

Portuguese stop consonants (total of 24 target consonants),

distributed as the following: ¡°p¡± ? Papai olha a pipa (Daddy

sees the kite): 4 target consonants; ¡°b¡± ?A Bibi babou (Bibi

drooled): 4 target consonants; ¡°t¡± ? O tatu ¨¦ teu (The armadillo is yours): 3 target consonants; ¡°d¡± ? O dedo da Duda

doeu (Duda? s ?nger hurt): 6 target consonants; ¡°k¡± ? O cuco

caiu aqui (The cuckoo clock fell here): 4 target consonants; and

¡°g¡± ? O Gugu ¨¦ legal (Gugu is cool): 3 target consonants. The 6

sentences are part of the Brazilian cleft articulation screening sentences, which consists of 23 sentences, each with a

single target consonant.

? 2023. Funda??o Otorrinolaringologia. All rights reserved.

Influence of an Intensive Speech Therapy Program

Speech samples were recorded using a Shure PG30

condensed/unidirectional head microphone (Shure, Niles,

IL, USA), positioned at  5 cm from the mouth, in an Intel

Pentium 4 (256MB HD, 15MB RAM) computer. The ?les were

recorded with wave extension using a Creative Audigy II

soundcard (Creative Technology Ltd., Jurong East,

Singapore), with the Sony Sound Forge 8.0 software (Sony

Corp. Tokyo, Japan), with a sampling rate of 44,100Hz, single

channel, 16 bits. To obtain the audio recordings, each patient

sat in a comfortable chair in a sound-isolated room in the

Phonetic Laboratory. The patients repeated each stimulus

sentence after the speech language pathologist (SLP).

Listening Material

The selected samples were edited using the Sound Forge 8.0

software, randomly distributing the recordings (obtained

pre- and post-ISTP) into a material presented to three SLPs

for a rating task of the 6 phrases analyzed in the present

study, 1 for each of the 6 consonants of interest (p, b, t, d, k,

and g).

Each listener rated individually the presence and absence

of glottal stop articulation in 888 target consonants of the

pre- and post-ISTP audio recorded sentences produced by the

37 patients (37 patients  24 target consonants ? 888 target

consonants pre-ISTP and 888 target consonants post-ISTP).

Listeners

Three female Brazilian certi?ed SLPs (listeners), different

from those who applied the therapy, with a minimum of

6 years of experience with management of cleft palate

speech, rated the samples to identify the presence of glottal

stops. The listeners were not aware of the purpose of the

present study nor were they familiar with any of the speakers. The SLPs self-reported having normal hearing.

Listening Task

The listeners received an AKG K414P headset earphone (AKG

Acoustics, Vienna, Austria) and a USB ?ash drive containing

the speech material. The material included a ?le with a

Microsoft PowerPoint (Microsoft Corp., Redmond, WA,

USA) presentation containing instructions for the rating

task and a ?le containing the randomly edited and anonymous recordings to be rated by the SLPs. The listeners

reviewed the instructions to become familiar with the rating

task. Twenty sentences produced by patients with a history

of cleft palate representative of productions with and without glottal stop articulation were presented to the listeners

as reference samples. The listeners were also instructed on

how to use the form to record their rating. The form was

prepared by the ?rst author speci?cally for this purpose.

The listeners were instructed to rate individually the

samples indicating either the presence or the absence of

glottal stop articulation. They were also instructed to use

their own personal computer with the earphone provided by

the investigators to listen to the samples. They could adjust

the audio level as needed. The recordings could be listened as

many times as the listener deemed necessary until being able

to establish the rating of presence or absence of glottal stops.

Andrade et al.

Inter-rater Agreement

The Kappa index of agreement was used to measure

the degree of inter-rater agreement, for each target consonant, pre- and post-ISTP.

Interpretation of Kappa scores15: Poor ? Kappa < 0.00;

Slight ? Kappa 0.00¨C0.20; Fair ? Kappa 0.21¨C0.40; Moderate

? Kappa 0.41¨C0.60; Substantial ? Kappa 0.61¨C0.80; Almost

perfect ? Kappa 0.81¨C1.00.

Statistical Analysis

Only the samples rated identically by at least two of the three

listeners, pre- and post-ISTP, were used for analysis and

comparison. Data analysis was calculated using percentage.

The comparison of the occurrence of glottal stop pre- and

post-ISTP was calculated using the McNemar test, adopting a

signi?cance level of 5% (p < 0.05).

The comparison of the proportion improvement among

the six stop consonants was calculated using the chi-squared

test and proportions. The comparison of the proportion of

improvement of each target consonant within the six sentences was calculated using the Cochran test, adopting a

signi?cance level of 5% (p < 0.05).

Results

The inter-rater agreement and the ratings were compared

between the three raters for each target consonant, pre- and

post-ISTP (?Table 1).

Out of the 888 target consonant possibilities in the preISTP audio recordings, glottal stop articulation was rated to

be present in 325 (37%) of the samples. Out of those 325

(100%), 197 (61%) remained with this error, and 128 (39%) no

longer presented it, post-ISTP. The comparison of the occurrence of glottal stop among the six stop consonants, pre- and

post-ISTP, was statistically signi?cant only for the ¡°p¡± and ¡°k¡±

consonants (chi-squared test; p ? 0,014). See ?Fig. 1.

The comparison of the occurrence of glottal stop and postISTP was statistically signi?cant only for the target consonants ¡°p1,¡± ¡°p2,¡± ¡°p3,¡± ¡°p4,¡± ¡°t1,¡± ¡°k1,¡± ¡°k2,¡± and ¡°d6¡± (McNemar test; p < 0.05). See ?Table 2.

The comparison of the proportion improvement among

the six stop consonants was statistically signi?cant for the p

consonant in relation to the k, b, d, and g consonants, and for

the t consonant in relation to the b, d, and g consonants (chisquared test; p < 0.05). The comparison of the proportion of

improvement of each target consonant within the six sentences was not statistically signi?cant (Cochran test;

p > 0.05).

Discussion

The aim of the present study was to investigate the in?uence

of an ISTP to correct glottal stop articulation in patients with

cleft palate. The results showed a decrease of consonants

with glottal stop after therapy, which is in agreement with

previous studies that investigated the ef?cacy of an ISTP for

cleft palate speech.12,13,16,17 Brazilian studies that compared

speech outcomes before and after therapy of individuals with

International Archives of Otorhinolaryngology

Vol. 27

No. 1/2023

? 2023. Funda??o Otorrinolaringologia. All rights reserved.

5

6

Influence of an Intensive Speech Therapy Program

Andrade et al.

Table 1 Inter-rater agreement percentage and Kappa values

for all 24 consonant targets, pre- and post-intensive speech

therapy program

Consonant

Pre-ISTP

Post-ISTP

% of

agreement

Kappa

% of

agreement

Kappa

p1

77

0.54

78

0.29

p2

78

0.58

71

0.21

p3

84

0.69

77

0.27

p4

75

0.51

73

0.25

t1

88

0.74

78

0.39

t2

93

0.86

73

0.30

t3

87

0.74

71

0.35

k1

91

0.82

78

0.50

k2

91

0.82

75

0.46

k3

91

0.82

68

0.37

k4

86

0.71

69

0.38

b1

95

0.83

84

0.33

b2

95

0.83

84

0.33

b3

93

0.73

80

0.12

b4

91

0.68

84

0.18

d1

86

0.64

86

0.40

d2

86

0.64

84

0.36

d3

86

0.64

84

0.37

d4

87

0.68

86

0.42

d5

89

0.73

84

0.29

d6

84

0.56

89

0.42

g1

91

0.74

91

0.69

g2

91

0.74

91

0.69

g3

84

0.51

78

0.31

Abbreviation: ISTP, intensive speech therapy program.

cleft palate also found a signi?cant reduction in the occurrence of CAs.12,18¨C21

Among the 36 patients who presented glottal stop articulation pre-ISTP, 5 (14%) were able to eliminate this error in all

target consonants; 4 (11%) did not show any change, which

means that they continued to present this error in the same

target consonants, and 27 (75%) presented a reduction in the

occurrence of this error post-ISTP. It is noteworthy that the

patients who remained with glottal stop articulation postISTP were referred to participate in the next ISTP module of

the Hospital of Rehabilitation of Craniofacial Anomalies or to

continue speech therapy elsewhere.

Our results have also shown that, among the six target

consonants with glottal stop articulation, the ¡°p¡± consonant

was the easiest to be corrected when compared with the ¡°k¡±

consonant. This can be explained by the fact that the ¡°p¡±

consonant, as an anterior and bilabial consonant, has visual

features easier to be learned using the facilitating cues,18,22

compared with the ¡°k¡± consonant, which is produced in the

International Archives of Otorhinolaryngology

Vol. 27

No. 1/2023

back of the mouth, where visual features are dif?cult to see.

Pinto (2016)20 also found in a retrospective study that the ¡°p¡±

consonant was the easiest consonant to be learned by the

cleft palate patients undergoing intensive speech therapy.

Despite the variation in the number of occurrences of

glottal stops in all target stop consonants pre-ISTP, it was

observed that the greater occurrence of glottal stops was

present in the voiceless consonants, when compared with

the voiced ones. Voiceless high-pressure consonants require

a greater amount of air pressure than the voiced ones, and

this may explain the vulnerability of the voiceless consonants to be produced with glottal stop.5 In general, most

target stop consonants (unvoiced and voiced) of the present

study have shown reduction in the number of glottal stop

occurrences, regardless of their position within the sentence,

although the unvoiced ones were those that signi?cantly

improved the most. This ?nding shows the ability of the

individual to generalize the correct consonant production by

using correct therapeutic strategies23,24 and approaches.25

The ?ndings of our study have also shown that all target

consonants presented a reduction of the occurrence of glottal

stop articulation regardless of their position within the

sentence, with the exception of the consonants ¡°d2¡± (O

dedo da Duda doeu) and ¡°d6 ¡± (O dedo da Duda doeu), which

presented a statistically signi?cant difference. This can be

explained by the generalizing ability of the patient to use the

correct therapeutic strategy to produce the target sound

wherever it appears in the word.26 Some studies show that

the generalization process can occur in other positions of the

word, in which the patient learns to produce a phoneme in a

certain position and proceeds to perform it correctly in other

positions.27,28

Good speech outcome of patients with cleft lip and palate

can be achieved either by intensive speech therapy or

conventional therapy, although studies suggest that in ISTPs,

the improvement can be achieved in a shorter period of time,

at a lower cost.13,14 Unlike conventional therapy, which is

usually based on one or two sessions per week with no ?xed

time to complete, intensive speech therapy, although based

on programmed modules, varies in the number of sessions,

on the duration of one module, and on the duration of the

therapy session. Intensive speech therapy programs with

modules of up to 2 months, with daily sessions ranging from

3 to 7 days a week, with  1times a day, have been reported in

the literature.20,21,29¨C31

Studies show that most speech therapists use the phonetic approach to treat individuals with cleft lip and palate, with

good results.32 However, some authors suggest that the

phonological approach can also be successfully used for

patients presenting with many CAs,25,33,34 highlighting

that future studies should be done to compare the results

between patients undergoing both approaches.

In the present study, 72% of the participants used a

pharyngeal bulb prosthesis (either temporary or permanent)

due to hypodynamic velopharynx. Studies have shown that

some individuals can improve the movement of their velopharyngeal structures with the use of the bulb by itself,12,35¨C39 and others would only accomplish better

? 2023. Funda??o Otorrinolaringologia. All rights reserved.

Influence of an Intensive Speech Therapy Program

Andrade et al.

Fig. 1 Distribution of the number of glottal stop occurrences for each of the six stop consonants and post-intensive speech therapy program

Table 2 Distribution of the presence of glottal stops among the 24 target consonants and post-intensive speech therapy program,

for the 37 patients

Target consonant

p1 (Papai olha a pipa)

Presence of glottal stop

Pre-ISTP

Post-ISTP

Difference

16 / 37

06 / 37

10

p2 (Papai olha a pipa)

17 / 37

07 / 37

10

p3 (Papai olha a pipa)

20 / 37

07 / 37

13

p4 (Papai olha a pipa)

18 / 37

08 / 37

10

t1 (O tatu ¨¦ teu)

20 / 37

10 / 37

10

t2 (O tatu ¨¦ teu)

18 / 37

11 / 37

07

t3 (O tatu ¨¦ teu)

22 / 37

14 / 37

08

k1 (O cuco caiu aqui)

20 / 37

12 / 37

08

k2 (O cuco caiu aqui)

21 / 37

14 / 37

07

k3 (O cuco caiu aqui)

17 / 37

16/ 37

01

k4 (O cuco caiu aqui)

21 / 37

15 / 37

06

b1 (A Bibi babou)

07 / 37

05 / 37

02

b2 (A Bibi babou)

07 / 37

05 / 37

02

b3 (A Bibi babou)

06 / 37

03 / 37

03

b4 (A Bibi babou)

06 / 37

04 / 37

02

d1 (O dedo da Duda doeu)

12 / 37

07 / 37

05

d2 (O dedo da Duda doeu)

11 / 37

07 / 37

04

d3 (O dedo da Duda doeu)

11 / 37

07 / 37

04

d4 (O dedo da Duda doeu)

11 / 37

08 / 37

03

d5 (O dedo da Duda doeu)

10 / 37

07 / 37

03

d6 (O dedo da Duda doeu)

12 / 37

04 / 37

08

(Continued)

International Archives of Otorhinolaryngology

Vol. 27

No. 1/2023

? 2023. Funda??o Otorrinolaringologia. All rights reserved.

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